Medical management of an ovarian ectopic pregnancy: a case ... · CASE REPORT Open Access Medical...

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CASE REPORT Open Access Medical management of an ovarian ectopic pregnancy: a case report Ozer Birge 1* , Mustafa Melih Erkan 2 , Ertugrul Gazi Ozbey 3 and Deniz Arslan 3 Abstract Background: Primary ovarian ectopic pregnancy is a rare type of ectopic pregnancy which has an estimated prevalence ranging from 1:7000 to 1:70,000 accounting for almost 3 % of all ectopic cases. Here we report the case of a 25-year-old woman who presented to our clinic with abdominal pain, 6 weeksdelay of menstruation and 3 days of vaginal bleeding, whose transvaginal ultrasonography showed an ectopic gestational sac with yolk sac inside, in her right ovary. This case shows that early diagnosis is very important particularly in places like the Sub-Saharan region of Africa. Case presentation: A 25-year-old African woman was referred to our clinic with 6 weeksdelay of menstruation, frequent increasing abdominal pain and 3 days of vaginal bleeding. Her general condition was good and her vital signs were normal. She felt tenderness in an abdominal examination and had a small amount of vaginal bleeding. Transvaginal ultrasonography showed an ectopic gestational sac with yolk sac inside, in her right ovary. Our final diagnosis was ectopic ovarian pregnancy and we successfully treated her with methotrexate. After 3 weeks of methotrexate administration her beta human chorionic gonadotropin was negative and a sonographic examination was completely normal. Conclusions: Ectopic ovarian pregnancy is a very important medical situation. It should be diagnosed in its early stages otherwise it could be life-threatening and surgical treatment may be inevitable. Because of the importance of fertility, medical treatment is an acceptable option and can be feasible with early diagnosis. Keywords: Ectopic pregnancy, Methotrexate, Ovarian pregnancy Background Primary ovarian ectopic pregnancy is a rare type of ectopic pregnancy which has an estimated prevalence ranging from 1:7000 to 1:70,000 accounting for almost 3 % of all ectopic cases [1]. It is usually terminated by a rupture in the first trimester and because of the increased vascularization of the ovarian tissue it leads to internal hemorrhage and hypovolemic shock status. The diagnosis is usually made by emergency laparotomies and histopathologic assessment. Diagnosis is made using the Spiegelberg criteria [2] which include: a. The gestational sac is located in the region of the ovary. b. The ectopic pregnancy is attached to the uterus by the ovarian ligament. c. Ovarian tissue in the wall of the gestational sac is proved histologically. d. The tube on the involved side is intact. Non-tubal pregnancies are the most common type of ectopic pregnancy and ovarian pregnancies are the second most common type; ovarian pregnancies are very common with intrauterine devices (IUDs). Surgical treatments are often performed in these cases because of the late onset of clinical symptoms which leads to late diagnosis [1, 2]. Methotrexate (MTX) treatment can be used for patients in the early phases if their condition is stable. * Correspondence: [email protected] 1 Nyala Sudan Turkey Training and Research Hospital, Department of Gynecology and Obstetrics, West Alezza District, Southern Nyala, Darfur, Sudan Full list of author information is available at the end of the article © 2015 Birge et al. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Birge et al. Journal of Medical Case Reports (2015) 9:290 DOI 10.1186/s13256-015-0774-6

Transcript of Medical management of an ovarian ectopic pregnancy: a case ... · CASE REPORT Open Access Medical...

Page 1: Medical management of an ovarian ectopic pregnancy: a case ... · CASE REPORT Open Access Medical management of an ovarian ectopic pregnancy: a case report Ozer Birge1*, Mustafa Melih

CASE REPORT Open Access

Medical management of an ovarian ectopicpregnancy: a case reportOzer Birge1*, Mustafa Melih Erkan2, Ertugrul Gazi Ozbey3 and Deniz Arslan3

Abstract

Background: Primary ovarian ectopic pregnancy is a rare type of ectopic pregnancy which has an estimatedprevalence ranging from 1:7000 to 1:70,000 accounting for almost 3 % of all ectopic cases. Here we report thecase of a 25-year-old woman who presented to our clinic with abdominal pain, 6 weeks’ delay of menstruation and3 days of vaginal bleeding, whose transvaginal ultrasonography showed an ectopic gestational sac with yolk sacinside, in her right ovary. This case shows that early diagnosis is very important particularly in places like theSub-Saharan region of Africa.

Case presentation: A 25-year-old African woman was referred to our clinic with 6 weeks’ delay of menstruation,frequent increasing abdominal pain and 3 days of vaginal bleeding. Her general condition was good and her vitalsigns were normal. She felt tenderness in an abdominal examination and had a small amount of vaginal bleeding.Transvaginal ultrasonography showed an ectopic gestational sac with yolk sac inside, in her right ovary. Our finaldiagnosis was ectopic ovarian pregnancy and we successfully treated her with methotrexate. After 3 weeks ofmethotrexate administration her beta human chorionic gonadotropin was negative and a sonographic examinationwas completely normal.

Conclusions: Ectopic ovarian pregnancy is a very important medical situation. It should be diagnosed in its earlystages otherwise it could be life-threatening and surgical treatment may be inevitable. Because of the importanceof fertility, medical treatment is an acceptable option and can be feasible with early diagnosis.

Keywords: Ectopic pregnancy, Methotrexate, Ovarian pregnancy

BackgroundPrimary ovarian ectopic pregnancy is a rare type ofectopic pregnancy which has an estimated prevalenceranging from 1:7000 to 1:70,000 accounting for almost3 % of all ectopic cases [1]. It is usually terminated by arupture in the first trimester and because of theincreased vascularization of the ovarian tissue it leads tointernal hemorrhage and hypovolemic shock status. Thediagnosis is usually made by emergency laparotomiesand histopathologic assessment.Diagnosis is made using the Spiegelberg criteria [2]

which include:

a. The gestational sac is located in the region of theovary.

b. The ectopic pregnancy is attached to the uterus bythe ovarian ligament.

c. Ovarian tissue in the wall of the gestational sac isproved histologically.

d. The tube on the involved side is intact.

Non-tubal pregnancies are the most common type ofectopic pregnancy and ovarian pregnancies are thesecond most common type; ovarian pregnancies are verycommon with intrauterine devices (IUDs). Surgicaltreatments are often performed in these cases because ofthe late onset of clinical symptoms which leads to latediagnosis [1, 2]. Methotrexate (MTX) treatment can beused for patients in the early phases if their condition isstable.

* Correspondence: [email protected] Sudan Turkey Training and Research Hospital, Department ofGynecology and Obstetrics, West Alezza District, Southern Nyala, Darfur,SudanFull list of author information is available at the end of the article

© 2015 Birge et al. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Birge et al. Journal of Medical Case Reports (2015) 9:290 DOI 10.1186/s13256-015-0774-6

Page 2: Medical management of an ovarian ectopic pregnancy: a case ... · CASE REPORT Open Access Medical management of an ovarian ectopic pregnancy: a case report Ozer Birge1*, Mustafa Melih

Case presentationA 25-year-old African obese woman with a history oftwo cesarean sections was referred to our clinic with6 weeks’ delay of menstruation, frequent increasing ab-dominal pain and 3 days of vaginal bleeding. She had aregular menstrual period before the symptoms. In hermedical history there was no record of use of an IUD,endometriosis or pelvic inflammatory disease. This washer third spontaneous pregnancy and there was no abor-tion. Her general condition was good and her vital signswere normal: blood pressure 110/70 mmHg, pulse 70beats per minute (bpm), temperature 36.5 °C. A physicalexamination showed minimal tenderness in all sides ofher abdomen with an increase in right lower pelvic sec-tion. A speculum examination showed a small amountof cervical bleeding, a palpable mass in rectouterine cav-ity and increased temperature and tenderness at rightadnexial region. Transvaginal ultrasonography (USG)showed empty uterine cavity with 11 mm thickness.However, her rectouterine cavity was observed to befilled with heterogenous liquid including septations andhyperechogenic areas which were thought to be a coagu-lum. An ectopic gestational sac and yolk sac seemed tobe inside her right ovary, and were identified close tothe midline, which correlated with her 6 weeks’ delay ofmenstruation (Fig. 1). The fetus and fetal heart beat werenot clearly seen. Vascular proliferation called ‘ring of fire’which is typical for ectopic ovarian pregnancy was de-tected around the gestational sac (Fig. 1). Her left ovaryand tubal structures seemed to be normal. She declaredher previous menstrual periods were regular but that herlast period was 2 months ago. Laboratory analysisshowed a white blood cell count (WBC) of 11,600/mm3,red blood cell count (RBC) of 400000/mm3, hemoglobin(Hb) of 12.3 g/dl, hematocrit (Htc) of 36 %, beta humanchorionic gonadotropin (HCG) of 6580 and normalurine results. She was diagnosed as having an ectopicovarian pregnancy and was hospitalized. She and herfamily were informed about the stability of the conditionand in view of her history of two previous cesarean sec-tions, medical treatment of MTX was planned. A singledose of 90 mg intramuscular MTX was administered.She was stable. A progressive decrease in her beta HCGlevels (4310 at fourth day, 2190 at seventh day, 210 at14th day) as well as a diminishing of intraabdominal li-quid and significant regression of her right ovarian sacwere observed and she was discharged with weekly betaHCG test control advice. At the third week after theMTX treatment her beta HCG level was below 5 andher intraabdominal fluid had nearly disappeared (Fig. 2).

DiscussionThe history of ectopic pregnancy is as old as humanity. Thefirst successful operation for ectopic pregnancy took place

Fig. 1 Ultrasound images of ovarian gestational sac before medicalmethotrexate therapy. (a) Ovarian gestational sac (b) Colour dopplerimage of gestational sac

Fig. 2 Control ultrasonographic image after the methotrexatetreatment

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in 1759 in the USA but the usual treatment was still medicalup to the 1800s with a maternal mortality rate reaching upto 60 % [3]. The high mortality rates drew special atten-tion which led to crucial developments in the diagno-sis and treatment of this condition. Salpingectomy,which started to be performed from the 1800s, is ob-served to be lifesaving because it decreased the ma-ternal mortality rates to nearly 5 %.Ovarian ectopic pregnancy is a rare variant of ectopic

pregnancy [4]. It occurs by fertilization of an ovumretained in the peritoneal cavity leading to implantationon the ovarian surface [5]. Women with ovarian ectopicpregnancies usually present with lower abdominal pain,menstrual irregularities as in other ectopic conditionsand corpus luteum cyst. Although early diagnosis andearly treatment are crucial, preoperative and sometimesintraoperative diagnoses are difficult. Diagnosis is usuallymade by pathological assessment and therefore the Spie-gelberg criteria are very important for the diagnosis ofectopic ovarian pregnancy [6].Prediagnosis is usually supported by increased beta

HCG levels. The current data inform that most casesoccur in the first trimester. Early onset rupture can leadto massive intraabdominal hemorrhage resulting inhypovolemia which can be life-threatening. Some rarecases that reach second trimester are also documented[7]. There are also published cases of twin ovarian ec-topic pregnancies and coincidence of uterine and ovar-ian ectopic pregnancies [7–10]. We also found somearticles on ectopic pregnancies of advanced gestationalage diagnosed preoperatively with USG and magneticresonance imaging (MRI) [7, 11]. In the study of Hallat,a preoperative diagnosis was achieved in 28 % of 25primary ectopic pregnancy cases. All other cases werediagnosed by pathological assessment postoperatively[12]. Phupong and Ultchaswadi declared that the evalu-ation of beta HCG together with transvaginal USG canbe helpful for early diagnosis [13].The cause of implantation anomalies in ovarian

ectopic pregnancy is not clear [7, 12, 13]. There are vari-ous hypotheses such as:

a. Delay of ovum liberation.b. Thickening of tunica albuginea.c. Tubal dysfunction.d. Intrauterine contraception devices (for example, IUDs).

Pelvic inflammatory disease does not have an effect onovarian ectopic pregnancy like it does on tubal pregnancy[9, 14]. IUDs are thought to be a main factor in ovarian ec-topic pregnancy cases according to the majority of studies.It is believed that IUDs trigger mild inflammation that dis-turbs the ciliary activity of the endosalpinx and leads toovum transport delay and ectopic implantation [15, 16]. In

our case, ectopic pregnancy was diagnosed from clinicaland laboratory examinations and evaluations of her condi-tion. Because of her two previous cesarean sections and thesuspicion of secondary salpingitis by endemic chronic pel-vic infections we performed medical treatment with MTX.Primary ovarian ectopic pregnancy is usually seen

among young fertile multipara women who use an IUD[17]. Berger and Blechner documented that the ratio ofovarian ectopic pregnancy among women using an IUDto all ectopic cases is 1:9; its prevalence in the generalpopulation is detected as 1:150 to 200 [16]. Our case hadno history of IUD usage. In the case series of Raziel et al.,18 of 20 cases of ovarian pregnancy were using an IUD[14]. The link between IUDs and ovarian pregnancy in fer-tile patients is worthy of comment. In their study, Lehfeldtet al. detected that the IUDs prevent uterine implantationby 99.5 % and tubal implantation by 95.5 %; however,there is no preventive effect on ovarian implantation [18].As the definitive diagnosis is made surgically and histo-

pathologically even in patients with early onset, surgicalinterventions have both a diagnostic and a therapeuticvalue. Because oophorectomy is a radical procedure forovarian ectopic pregnancy, consideration should be givento the patient’s age, fertility, her desire to have furtherpregnancies, and the size of the mass; wedge resection canalso be another surgical option.Medical and conservative treatments have also been in-

troduced in recent years to prevent ovarian tissue loss,pelvic adhesions and to preserve the patient’s fertility.These include administration of mifepristone for patientsdiagnosed using a transvaginal USG, parenteral prosta-glandin F2a and MTX treatment for non-ruptured casesdetected with laparoscopy [11, 19]. Pagidas and Frishmanperformed MTX treatment for ovarian ectopic cases diag-nosed using transvaginal USG and achieved healing. Theyemphasized that early staged cases diagnosed by transvagi-nal USG, can benefit from MTX treatment [20]. Di Luigiet al. also performed and succeeded with multidose MTXtreatment which they administered to a 37-year-old pa-tient with a history of two previous cesarean sections andIUD usage; she was diagnosed at 6 weeks of ectopic ovar-ian pregnancy by use of a transvaginal USG. They empha-sized that with careful clinical evaluation and transvaginalexamination early staged ovarian ectopic cases can betreated medically which preserves the normal anatomycrucial for fertility [21]. A review of the data shows thatMTX treatment is chosen after a clear diagnosis and de-tection of the localization of ectopic cases by laparoscopyand therefore laparoscopy is declared to be a supportingdiagnostic procedure [22]. In cases in which the gesta-tional sac is lower than 30 mm, without fetal cardiac activ-ity, and less than 6-weeks old, MTX treatment issupported in particular and is superior to surgery becauseit does not disturb fertility [23].

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In our case although she had pelvic fluid of hemorrhagiccharacter that could have been caused by pelvic rupture, aclinical evaluation and consideration of her previous oper-ations led us to treat her medically. Her beta HCG levelsprogressively decreased after single dose MTX and shedid not face the risks of further surgery.

ConclusionsAlthough ovarian ectopic pregnancy is a rare condition,after careful evaluation, the selection of medical proce-dures should take into consideration the preservation offertility particularly for young patients.

ConsentWritten informed consent was obtained from the patientfor publication of this case report and any accompanyingimages. A copy of the written consent is available forreview by the Editor-in-Chief of this journal.

AbbreviationsHCG: Human chorionic gonadotropin; IUD: Intrauterine device;MTX: Methotrexate; USG: Ultrasonography.

Competing interestsThe authors declare that they have no competing interests.

Authors’ contributionsBO, AD, and OEG were involved in management of the patient. BO and EMMdrafted the manuscript. EMM and BO provided valuable input and guidanceduring the preparation of the manuscript. BO was responsible for overallmanagement of the patient and supervised the writing critically. All authorsread and approved the final manuscript.

AcknowledgementsWe would like to thank the staff of Nyala Sudan Turkey Research andTraining Hospital, the patient and relatives who were kind enough to givetheir consent to publish this case report.

Author details1Nyala Sudan Turkey Training and Research Hospital, Department ofGynecology and Obstetrics, West Alezza District, Southern Nyala, Darfur,Sudan. 2Celal Bayar University Hospital, Department of Gynecology andObstetrics, Manisa, Turkey. 3Nyala Sudan Turkey Training and ResearchHospital, Department of Urology, Nyala, Darfur, Sudan.

Received: 6 July 2015 Accepted: 23 November 2015

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